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Forward-Looking Statements This presentation contains forward-looking statements within the meaning of the Private Securities Litigation Reform Act of 1995. Words such as "may," "will," "expect," "plan," "anticipate" and

Key Takeaway: Ovid Therapeutics Corporate Presentation February 2019 Exhibit 99.1 Forward-Looking Statements This presentation contains forward-looking statements within the meaning of the Private Securities Litigation Reform Act of 1995. Words such as "may," "will," "expect," "plan," "anti

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Ovid Therapeutics Corporate
Presentation February 2019 Exhibit 99.1
Forward-Looking Statements This
presentation contains forward-looking statements within the meaning of the Private Securities Litigation Reform Act of 1995. Words such as "may," "will," "expect," "plan," "anticipate" and similar expressions (as well as other words or expressions
referencing future events or circumstances) are intended to identify forward-looking statements. Forward-looking statements contained in this presentation include statements about the progress, timing, clinical development and scope of clinical
trials and the reporting of clinical data for the Company's product candidates; the potential clinical benefit of the Company's product candidates; the timing and outcome of discussions with regulatory authorities; the completion of
business development agreements; and the use of 24HC as a biomarker for target engagement. Each of these forward-looking statements involves risks and uncertainties. These statements are based on the Company's current expectations and
projections made by management and are not guarantees of future performance. Therefore, actual events, outcomes and results may differ materially from what is expressed or forecast in such forward-looking statements. Factors that may cause actual
results to differ materially from these forward-looking statements are discussed in the Company's filings with the U.S. Securities and Exchange Commission, including the "Risk Factors" sections contained therein. Except as otherwise required
under federal securities laws, we do not have any intention or obligation to update or revise any forward-looking statements, whether as a result of new information, future events, changes in assumptions or otherwise.
Ovid Highlights: Building a Leading
Company in Neurology Ovid is focused on developing novel medicines for patients with rare CNS disorders OV101 is entering a Phase 3 pivotal study in 2H 2019 Angelman Syndrome is a rare neurodevelopmental disorder for which no treatments exist End of
Phase 2 meeting with FDA guiding single pivotal Phase 3 strategy with ~60 patients Ongoing Phase 2 in Fragile X will yield data in 2H 2019 OV935 is in multiple Phase 2 studies for rare epilepsy types Significant market opportunity for novel,
effective treatments Phase 1b/2a data demonstrated positive signal indicative of continued advancement Global partnership with Takeda for OV935 Cost and profit sharing; Ovid leads development and commercialization in US/EU Leadership team with deep
experience developing drugs and building companies
Late preclinical- to
clinical-stage assets Clear Strategy That Enables Scalable Model
Robust Product Pipeline in
Neurodevelopment and Epilepsies The company is considering a rationalization of its resources to focus on its clinical stage assets, OV101 and OV935, which may include elimination or consolidation of functions, including the elimination or reduction
of early stage research personnel. * Also known as TAK-935. Co-development program with Takeda Pharmaceutical Company Limited pursuant to a license and collaboration agreement. RESEARCH PRECLINICAL PHASE 1 PHASE 2 PHASE 3 -selective GABAA
receptor agonist Angelman Syndrome OV101 -selective GABAA receptor agonist Fragile X Syndrome OV101 Undisclosed Neurological disorders and epilepsy RESEARCH OV329 GABA aminotransferase inhibitor Treatment-resistant Epilepsy OV935*
Developmental and Epileptic Encephalopathies CH24H inhibitor CDKL5 deficiency disorder & Dup15q syndrome CH24H inhibitor Dravet syndrome & Lennox-Gastaut syndrome Angelman Syndrome: Orphan Drug & Fast Track Designations Fragile X
Syndrome: Orphan Drug & Fast Track Designations DEE: Orphan Drug Designations for LGS and Dravet Syndrome Plan to start pivotal Ph 3 NEPTUNE in 2H19
OV101 Potential First-in-Class
Therapeutic for Angelman & Fragile X Syndromes
OV101: Significant Opportunity in Two
Serious Rare Disorders Angelman syndrome Fragile X syndrome Well Defined Genetic Disorders No approved therapies in US, EU, or ROW; treatment is limited to supportive care Lifelong disorders that present early in childhood Disruption of behavior,
anxiety and cognitive functions Most common inherited form of intellectual disability Prevalence**: ~1/3,600-4,000 males ~1/4,000-6,000 females Global disruption in behavior, motor, sleep and cognitive functions Prevalence in general population*: 1
in 12,000 to 20,000 people . Sources: *NORD, NIH website, Genetics Home Reference and NCBI . ** National Fragile X Foundation
1Meera et al., J Neurophysiol. 2011;
2Belelli et al., J. Neurosci.2005; 3 Duguid et al. Journal of Neurosci. 2012 4Olmos-Serrano et al. J. Neurosci. 2010; 5Egawa et al. Sci. Trans. Med. 20126Olmos-Serrano et al. Dev. Neuro. 2011 Angelman syndrome and Fragile X syndrome result in
reduced GABA levels, leading to reduced tonic inhibition and subsequent emergence of symptoms Only -selective, extrasynaptic GABAA receptor agonist in clinical development Distinct from GABA allosteric modulators, as it functions when
endogenous GABA is deficient Potentiates tonic inhibition at low nM concentrations1,2 Restores tonic inhibition in Angelman and Fragile X syndrome mouse models4,5,6 OV101: First-in-Class GABAA Receptor Agonist with Potential to Restore Tonic
STARS is First Industry-Sponsored
Clinical Trial in Angelman Syndrome * Excluding, poorly controlled seizure activity, concomitant use of minocycline, levodopa, zolpidem, zaleplon, eszopiclone, ramelteon, or cannabinoid derivatives or any other investigational agent, device,
and/or procedure. ; BID=twice a day; BL=baseline; QD=every day. Trial Design and Key Inclusion Criteria: 88 adults and adolescents 12 sites in US, 1 site in Israel Age 13-49 years, inclusive Molecular confirmation of Angelman Receiving a
stable regimen of concomitant medications for at least 4 weeks prior to baseline* Primary Outcome Measure: Safety and tolerability of OV101 vs placebo Exploratory Outcome Measures: Efficacy measures of OV101 vs placebo Placebo (placebo morning,
placebo night) OV101 BID (10 mg morning, 15 mg evening) OV101 QD (placebo morning, 15 mg evening) -2 0 6 12 Screening Baseline & Randomize (1:1:1) Assessment Points (weeks)
Four discontinued due to AEs
(placebo, n=1 (irritability), QD, no discontinuations, BID, n=3 (myoclonus, seizure, irritability/anxiety/sleep disorder) At least one treatment-emergent AE: placebo (25/29 - 86.2%), 15 mg QD (27/29 --93.1%) and BID (25/29 - 86.2%) There
were no deaths across OV101 and placebo arms STARS Trial: OV101 Achieved Primary Endpoint of Safety and Tolerability as Measured by Incidence of Adverse Events 1Safety set: All subjects who received at least one dose of study drug. *Descriptive
data. Drug-related TEAEs (n=4) AE=adverse event; BID=twice a day; QD=once a day; SAE=serious adverse event; TEAE=treatment-emergent adverse event Source: AACAP Poster 2018. Majority of AEs were mild Discontinuations due to AEs were low 2
patients experienced SAEs of worsening seizures 1 in QD - not related 1 in BID - possibly related Incidence n (%)* Placebo (n=29) OV101 QD (n=29) OV101 BID (n=29) Fever 2 (6.9) 7 (24.1) 1 (3.4) Rash 1 (3.4) 3 (10.3) 2 (6.9) Seizure 0 2
(6.9) 3 (10.3) Enuresis 0 2 (6.9) 1 (3.4) Myoclonic epilepsy 0 1 (3.4) 2 (6.9) Otitis media 0 2 (6.9) 1 (3.4) Viral infection 0 1 (3.4) 2 (6.9) Overall, OV101 was well tolerated with a favorable risk profile AEs Occurring More Frequently in
OV101 Treatment Groups vs. Placebo1
Clinical Global Impressions -
Improvement (CGI-I) is a Common Outcome Measure used to Assess Overall Clinical Symptomatology * Scores are assessed from baseline, defined as initiation of treatment. AS = Angelman syndrome ** a framework will be developed for study investigators
to ensure uniform use of the validated CGI-I scale Sources: AACAP Poster 2018 and Ovid press release dated December 6, 2018 Patients Rated on a 7-Point Scale* 1 = very much improved 2 = much improved 3 = minimally improved 4 = no change 5 =
minimally worse 6 = much worse 7 = very much worse Individuals have different degrees of difficulty with sleep, communication, behavior, motor, epilepsy and other symptoms: Even when they share the same mutation of the UB3a gene CGI-I is a tool used
to measure changes in these patient individually A clinically meaningful change in an individual could include improvements in any or all of these areas: Sleep (e.g. reduced daytime sleepiness & latency to sleep onset) Behavior (e.g. increased
vocalizations & awareness) Motor function (e.g. gross motor skills, walking more steadily or fine motor skills such as improved grip) What CGI-I Means to an Individual Established, validated, physician-reported measure Used in neurology and
psychiatry trials Captures totality of symptoms, including improvement in global neurological deficit Currently, no existing AS-specific scales or assessment instruments Plan to use CGI-I as primary efficacy endpoint due to rare nature, lack of
treatment options, heterogeneity and lack of AS-specific instruments**
STARS Data Suggest Changes in Sleep
and Motor Contributed to the Statistically Significant Improvement in CGI-I Global Improvement CGI-I Sleep Actigraphy Clinical Impression -sleep Motor Bayley PEDI-CAT CHAQ Zeno Walkway Exploratory Endpoints: Observed Changes CGI-I symptoms
overall (MMRM1) = (QD) p=0.0006; (BID) Not significant; (combined) p=0.0103 Sleep Domain (15 mg QD dose) Latency to sleep onset - Diff=-25.7 minutes, P=0.0147 Clinical impressions of sleep - Diff=-0.77, p=0.0141 Motor Domain (15 mg QD
dose) BSID-III (post-hoc, responder analysis) -Changes in overall motor response (54%, p=0.0889) and gross motor only (36% p=0.0522) PEDI-CAT (post hoc) mobility score (p=0.0935; per protocol p=0.0475), and daily activity score (p=0.1170); per
protocol (p=0.0869) CHAQ -(p=0.0704) ZenoTM Walkway - reductions in mean cadence (p=0.0340) and stride velocity (p=0.0406) 1MMRM = Mixed Model Repeated Measure; 2Bayley Scales of Infant and Toddler Development Third Edition 3rd Edition
Source: AACAP Poster 2018
Response Based on CGI-I Symptoms
Overall and Clinical Impression of 9 Domains at Week 12* 100 90 80 70 60 50 40 30 20 10 0 Proportion of subjects (%) Placebo (n=28) Combined OV101 (n=57) STARS Trial: OV101 Reached Statistical Significance in CGI-I at Week 12 27.4% difference
p=0.0206 66.7% 39.3% Based on Fisher's exact test responder analysis. CGI-I=Clinical Global Impressions - Improvement A subject is defined as a responder if any of his/her clinician assessed CGI-I items scores is less than or equal to 3
Source: AACAP Poster 2018.
Data provide supportive rationale
for OV101 15 mg QD as an efficacious dose in AS STARS Trial: CGI-I Symptoms Overall Scores Compared with Placebo Based on Mixed Model Repeated Measures (MMRM) analysis including fixed effects for visit, treatment, age (adult vs adolescent) and visit
by treatment interaction. MMRM is defined as the analysis used for imputing missing data. AS=Angelman syndrome; BID=twice a day; CGI-I=Clinical Global Impressions - Improvement; QD=every day; SD=standard deviation. Source: AACAP Poster 2018
*LS mean difference [Drug-Placebo] (95% CI) was -0.78 (-1.22, -0.35) with OV101 QD and -0.21 (-.064, .022) with OV101 BID p=0.3446 p=0.0006 Patients in each CGI-I score category (%) CGI-I Symptoms Overall at Week 12 Mean CGI-I Symptoms Overall Score
STARS Phase 2 Trial : Summary and
Conclusions Source: Ovid press release dated December 6, 2018 STARS achieved primary endpoint of safety and tolerability OV101 was well-tolerated with an overall favorable safety profile Global function Statistically significant improvement in CGI-I
in the OV101 QD treatment group Sleep Reduction in sleep onset latency (as measured by actigraphy) and improvement in overall sleep (as measured by clinical impression of sleep domain) in the OV101 15 mg QD treatment group vs. placebo Sleep
dysfunction is clinically relevant reduction in Angelman syndrome, and the observed reduction in latency to sleep onset may be indicative of target engagement Motor Motor domain changes noted in the BSID-III, PEDI-CAT, CHAQ Disability Index, and
Zeno Walkway Behavior The PGI reported improvements in communication, challenging behavior, and anxiety among patients who showed clinically meaningful improvement in CGI-I. However, no significant differences were found on the ABC-C and
OV101's Development in
Angelman Syndrome - Next Steps Pivotal Phase 3 NEPTUNE trial Design based on End-of Phase 2 Meeting with the FDA Single 12-week trial, two-arm, randomized, double-blind, placebo-controlled trial Once-daily dose Approximately 50-60 patients
aged 4 to 12 years Primary endpoint of change in overall CGI-I score Trial initiation pending FDA concurrence on the study protocol and supporting framework and materials Enrollment expected to begin in 2H:19 ELARA trial, an open-label extension
study First patient dosed for open-label extension study expected in 1Q:19 Source: Ovid press release dated December 6, 2018
OV101 in Fragile X Syndrome: Phase 2
Clinical Trials Underway Orphan Drug and Fast Track designations Primary Objective: Assess safety and tolerability of OV101 over 12 weeks of treatment across different daily dosing regimens Patient Population: Adult and adolescent males with
diagnosis of FXS (n=30) Ages: 13-22 years Timeline: Trial initiated in 3Q18, data expected in 2H:19 Endpoints: Primary - incidence of AEs. Secondary - changes in behavior Primary objective: Non-drug study to assess suitability of scales
to measure behavior, sleep, and functioning in individuals with Fragile X syndrome 0
OV935 New Approach to Potentially
Treat Rare Developmental and Epileptic Encephalopathies
Refractory seizures do not respond
to medicines that exert anti-seizure effect through same target or MOA as marketed AEDs Approximately 100,000- 200,000 US Population with Rare Seizure Disorders* Illustrative purposes only 1 out of 3 PATIENTS Small number achieve seizure freedom
with addition of 1 or more AEDs 1 out of 2 PATIENTS ALL EPILEPSY PATIENTS Current marketed AEDs act through four main mechanisms: 1. Modulation of voltage-gated ion channels 2. Enhancement of GABA-mediated inhibition 3. Interactions with elements of
the synaptic release machinery 4. Blockage of ionotropic glutamate receptors Seizure free with one AED Refractory seizures despite available medications Novel Mechanism of Actions are Needed to Treat Refractory Seizures Source: *Orphanet
Robust Preclinical Data Across
Several Well-Established Models on Potential Anti-Epileptogenesis Mechanism of OV935 Survival benefit in two transgenic epilepsy models Anti-inflammatory and glial modulatory activity contributing to disease modification in animal models
Neuroprotection from a certain type of glutamate toxicity OV935 Preclinical and non-clinical findings suggest potential of:
CH24H is a brain-specific enzyme for
cholesterol catabolism 24HC is a clinically monitorable plasma marker that appears to correlate with active target engagement OV935: Mechanism of Action OV935 T In Plasma In Brain *CH24H, cholesterol 24 hydroxylase ; 24HC,
24S-hydroxycholesterol CH24H Pathway is Unique to the Brain - 24HC is a Measurable Marker in Plasma*
l. J May address underlying
mechanisms behind refractory seizures in epilepsy May provide benefit through pathways that are not targets of conventional anti-epileptic drugs (AEDs) Orphan Drug designations for Dravet and Lennox- Gastaut syndromes Composition-of-matter patents
through 2032, without regulatory extensions OV935 Could Break the Vicious Cycle CH24H OV935 1Ba rker-Haliski et al.,Cold Spring Harb Perspect Med. 2015 2Ma s t N, et a l. J Biol Chem. 2017 3Sodero AO, et a l. EMBO J. 2012 4Pa ul SM, et al. J
Neurosci. 2013 5Sun MY, et a Neurophysiol. 2016 6Li ns enbardt AJ, et al. Neuropharmacology. CH24H Sei zures The Vicious Cycle OV935: First in Class Inhibitor of CH24H
Single doses up to 1,350mg well
tolerated; no SAEs reported In 14-day MAD* trial, doses of 100mg, 300mg and 400mg QD were well tolerated All TEAEs resolved with continued dosing through day 15 OV935 led to a dose dependent decrease in 24HC levels Correlation between plasma 24HC
and enzyme occupancy (PET) may inform dose selection OV935: Summary Results from Four Phase 1 Trials in Healthy Volunteers Dosing (days) % of Baseline plasma 24HC conc. *One volunteer at 600mg QD experienced acute psychosis; one volunteer at 300mg
BID experienced an event of confusional state; one placebo volunteer reported events of nightmares, spatial disorientation, insomnia and dizziness Results
Last updated: Feb 19, 2019